Infiltrating lipomatosis of the face (ILF) is a congenital nonhereditary disorder in which the tissues of the facial region are infiltrated for unencapsulated mature adipocytes. Most patients report facial changes at birth, typically with unilateral involvement, affecting soft and bone tissues, as well as dental alterations. Multiple surgeries and recurrences are not uncommon events. Recent studies suggest that ILF belongs within the PIK3CA-related overgrowth spectrum.

A 12-year-old female patient was referred for evaluation of dental occlusion. Extraoral examination revealed slight right facial asymmetry. Intraoral examination showed that the occlusion plane was inclined downwards on the right side. Moreover, early tooth eruption, as well as a nodular swelling in the hard palate, on the right side, was visualized.

Imaginological exams of palatal lesion supported a disorder than neoplastic process. After palate biopsy, the diagnosis was ILF.

The current case expands the clinicopathological spectrum of ILF, with subtle facial involvement being possible, and highlights the importance of dentistry in diagnosing this disorder.